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An unusual cause of nasal airway obstruction in a neonate: trans-sellar, trans-sphenoidal cephalocoele

Published online by Cambridge University Press:  27 July 2011

R A Steven ,
M P Rothera ,
V Tang  and
I A Bruce
R A Steven*
Affiliation:
Department of Paediatric Otolaryngology, Royal Manchester Children's Hospital, Manchester, UK
M P Rothera
Affiliation:
Department of Paediatric Otolaryngology, Royal Manchester Children's Hospital, Manchester, UK
V Tang
Affiliation:
Department of Paediatric Otolaryngology, Royal Manchester Children's Hospital, Manchester, UK
I A Bruce
Affiliation:
Department of Paediatric Otolaryngology, Royal Manchester Children's Hospital, Manchester, UK
*
Address for correspondence: Dr R A Steven, Department of Paediatric OtolaryngologyRoyal Manchester Children's Hospital, Oxford Road, Manchester, United KingdomM13 9WL E-mail: [email protected]
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Abstract

Introduction:

Neonates are obligate nasal breathers, and nasal obstruction may have serious implications. We present an extremely rare cause of neonatal nasal obstruction, and its management.

Case report:

An eight-day-old neonate was referred for upper airway obstruction. Initial investigations had identified no obvious cause. Rigid airway endoscopy revealed a large, cystic lesion appearing to arise from the roof of the posterior nasal space. Computed tomography and magnetic resonance imaging indicated a basal cephalocoele projecting inferiorly into the oropharynx, with an intracranial connection to the pituitary fossa. Histology showed fibrovascular tissue lined on one aspect by respiratory type epithelium, with mucous glands present. The tissue contained multiple cystic spaces lined by choroid plexus epithelium, with glial tissue present in the walls of the mass. A transpalatal excision of the nasopharyngeal cephalocoele, with closure of the intracranial connection, palatal repair and lumbar drain placement was undertaken. Post-operative recovery was uneventful, with no evidence of cerebrospinal fluid leakage or palatal dysfunction.

Conclusion:

This surgical approach gave excellent access whilst avoiding the obvious morbidity associated with an intracranial approach. Nasal masses should be considered in the differential diagnosis of neonatal respiratory distress due to nasal obstruction.

Keywords

EncephaloceleMeningoceleInfant, NewbornAirway ObstructionOtorhinolaryngologic Surgical ProceduresCephaloceleTrans-sphenoidalTrans-sellar
Type
Clinical Records
Information
The Journal of Laryngology & Otology , Volume 125 , Issue 10 , October 2011 , pp. 1075 - 1078
Copyright
Copyright © JLO (1984) Limited 2011

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Footnotes

Presented at the 139th Semon Club, 21 May 2010, Guy's and St Thomas' Hospital, London, winning the Professor Leslie Michaels Award

References

1Shrier, DA, Wang, AR, Patel, U, Monajati, A, Chess, P, Numaguchi, Y. Benign fibrous histiocytoma of the nasal cavity in a newborn: MR and CT findings. AJNR Am J Neuroradiol 1998;19:1166–8Google Scholar
2Barkovich, AJ, Vandermarck, P, Edwards, MSB, Cogan, PH. Congenital nasal masses: CT and MR imaging features in 16 cases. AJNR Am J Neuroradiol 1991;12:105–16Google Scholar
3Puppala, B, Mangurten, HH, McFadden, J, Lygizos, N, Taxy, J, Pelletierre, E. Nasal glioma presenting as neonatal respiratory distress: definition of the tumor mass by MRI. Clin Pediatr 1990;29:49–52Google Scholar
4Kim, DW, Low, W, Billman, G, Wickersham, J, Kearns, D. Chondroid hamartoma presenting as a neonatal nasal mass. Int J Pediatr Otorhinolaryngol 1999;47:253–9CrossRefGoogle ScholarPubMed
5Patterson, K, Kapur, S, Chandra, RS. “Nasal gliomas” and related brain heterotopias: a pathologist's perspective. Pediatr Pathol 1986;5:353–62CrossRefGoogle ScholarPubMed
6Lewin, ML. Sphenoethmoidal cephalocele with cleft palate: transpalatal versus transcranial repair. Report of two cases. J Neurosurg 1983;58:924–31Google Scholar
7Kennedy, EM, Gruber, DP, Billmire, DA, Crone, KR. Transpalatal approach for the extracranial surgical repair of transsphenoidal cephaloceles in children. J Neurosurg 1997;87:677–81CrossRefGoogle ScholarPubMed
8Mylanus, EA, Marres, HA, Vlietman, J, Kollee, LA, Freihofer, HP, Thijssen, HO et al. Transsellar sphenoidal encephalocele and respiratory distress in a neonate: a case report. Pediatrics 1999;103:e12Google Scholar
9Suwanwela, C, Suwanwela, N. A morphological classification of sincipital encephalomeningoceles. J Neurosurg 1972;36:201–11Google Scholar
10Kahyaoglu, O, Cavusoglu, H, Musluman, AM, Kaya, RA, Yilmaz, A, Sahin, Y et al. Transsellar transsphenoidal rhino-oral encephalocele. Turk Neurosurg 2007;17:264–8Google ScholarPubMed
11Blaivie, C, Lequeux, T, Kampouridis, S, Louryan, S, Saussez, S. Congenital transsphenoidal meningocele: case report and review of the literature. Am J Otolaryngol 2006;27:422–4Google Scholar
12Schick, B, Brors, D, Prescher, A. Sternberg's canal – cause of congenital sphenoidal meningocele. Eur Arch Otorhinolaryngol 2000;257:430–2CrossRefGoogle ScholarPubMed
13Raman Sharma, R, Mahapatra, AK, Pawar, SJ, Thomas, C, Al-Ismaily, M. Transsellar transsphenoidal encephalocele: report of 2 cases. J Clin Neurosci 2002;9:89–92Google Scholar
14Herman, P, Sauvaget, E, Guichard, JP, Tran Ba Huy, P. Intrasphenoidal transsellar encephalocele repaired by endoscopic approach. Ann Otol Rhinol Laryngol 2003;112:890–3CrossRefGoogle ScholarPubMed
15Gray, H. Anatomy of the Human Body. Twentieth Edition. Lea and Febiger, Philidelphia, 1918CrossRefGoogle Scholar
16Gursan, N, Aydin, M, Atlas, S, Ertas, A. Intranasal encephalocele: a case report. Turk J Med Sci 2003;33:191–4Google Scholar
17Castelnuovo, P, Bignami, M, Pistochini, A, Battaglia, P, Locatelli, D, Dallan, I. Endoscopic endonasal management of encephaloceles in children: an eight-year experience. Int J Pediatr Otorhinolaryngol 2009;73:1132–6CrossRefGoogle ScholarPubMed